Chondromyxoid Fibroma of Lumbar Vertebrae: Case Report And

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ISSN: 2643-4474 KAYA. Neurosurg Cases Rev 2020, 3:043 DOI: 10.23937/2643-4474/1710043 Volume 3 | Issue 2 Neurosurgery - Cases and Reviews Open Access

Case Report and Literature Review Chondromyxoid Fibroma of Lumbar Vertebrae: Case Report and Literature Review İsmail KAYA, MD* Check for Department of Neurosurgery, Ministry of Health, Şırnak State Hospital, Republic of Turkey updates *Corresponding author: İsmail Kaya, MD, Department of Neurosurgery, Şırnak State Hospital, Ministry of Health, Şirnak/ Turkey, Tel: 054-692-573-40 long bone metaphysis. Jaffe and Lichtenstein first de- Abstract scribed in 1948 [1]. Although, chondromyxoid fibroma Objective: Chondromyxoid fibroma is a benign tumor of is a relatively rare tumor spinal attachment is even rar- long bone metaphysis. In this article, we reported L4 laminar attachment of chondromyxoid fibroma case and made er. Chondromyxoid fibroma more frequently found cer- extensive literature revive. vical and thoracic segment. To our knowledge 11 case Methods: A 56-year-old Caucasian female unable to walk of lumbar chondromyxoid fibroma cases has described without pain at her right leg diagnosed with chondromyxoid until this case (Table 1) [2-10]. fibroma at right l4 lamina. In this article, we reported lumbar (L) 4 laminar at- Result: Complete excision of the lesion via routine lumbar tachment of chondromyxoid fibroma case and made ex- disc surgery with 6 months follow up without pain. Her Hy- tensive literature review. poesthesia cured also muscle weakness totally recovered. Conclusion: Chondromyxoid fibroma is rare bone tumor Material and Methods even rarer spinal involvement. Because of total removal can cure disease and partial removal has chance of malignant A 56-years-old Caucasian female was coming to our transformation we must consider chondromyxoid fibroma in clinic with unable to walk without pain at her right leg. our differential diagnosis. Patient didn’t describe neurologic claudication. Her Keywords complaints have been worsened for 6 months. Patient has no incontinence. Chondromyxoid fibroma, Lumbar vertebrae, Literature review Her physical exam showed right lasegue positivity at Abbreviations 30-degree, right extensor hallucis longus muscle weakness as much as 2/5. Also, right L5 dermatomal hypoes- Cmf: Chondromyxoid fibroma; L: Lumbar; MRI: Magnetic resonance imaging thesia. Plain X-ray showed no significant indication of chon- Highlights dromyxoid fibroma because of adherent structures and • Chondromyxoid fibroma must be considered for dif- superposition of multiple layers (Figure 1 and Figure 2). ferential diagnosis of low back pain. Magnetic resonance myelogram showed cerebrospi- • Extensive surgery provides good results with a very nal fluid flow obstruction on L4-5 disc space (Figure 3). low rate of recurrence. T1-weighted images showed low signal intensity adher- • More work must be put to understand nature of ent to right L4 lamina and foramina stenosis (Figure 4). Chondromyxoid fibromas. T2-weighted images showed high signal lesion with low signal intensity rim, which is very similar to neighbor Introduction bony structure (Figure 5 and Figure 6). Chondromyxoid fibroma (cmf) is a benign tumor of We suspected extrude disc herniation. An informed

Citation: KAYA I (2020) Chondromyxoid Fibroma of Lumbar Vertebrae: Case Report and Literature Review. Neurosurg Cases Rev 3:043. doi.org/10.23937/2643-4474/1710043 Accepted: September 29, 2020; Published: October 01, 2020 Copyright: © 2020 KAYA I. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

KAYA. Neurosurg Cases Rev 2020, 3:043 • Page 1 of 5 • DOI: 10.23937/2643-4474/1710043 ISSN: 2643-4474

1 2

Figure 1 and Figure 2: Plain X-ray (anteroposterior and lateral orientations).

3 4 Figure 3 and Figure 4: Magnetic resonance myelogram and T1-weighted sagittal magnetic resonance images. consent was obtained from patient in accordance the skin and subcutaneous tissue passed via 2 centimeters preoperative Turkish Neurosurgical Society consent of midline skin incision. Right paravertebral muscles form list and it was clearly stated that the data could be dissected using periosteal elevator. Right lamina of L4 used in pursuant of ethical rules. According to planned vertebrae totally removed and it was very soft purple surgery patient went under general anesthesia. Patient colored mass that pressing L5 root. Routinely curated carefully placed tilting operating table and positioned as bony margins. We suspected malignancy and sampled genupectorally. We used scopy to identify surgical area. it. There was no extrude disc material and posterior lon- After we made adequate cleaning and closure identified gitudinal ligament was intact. After foraminotomy per-

KAYA. Neurosurg Cases Rev 2020, 3:043 • Page 2 of 5 • DOI: 10.23937/2643-4474/1710043 ISSN: 2643-4474

5 6 Figure 5 and Figure 6: T2-weighted sagittal and axial magnetic resonance images.

formed hemostasis provided. Layers closed according to anatomical orientation. 2 gr cephazolin was used for 7 prophylactic purposes (Figure 7, Figure 8 and Figure 9). Results Histopathological diagnosis confirmed as chondromyxoid fibroma. Patient followed for 6 months without pain. Her hypoesthesia cured also muscle weakness totally recovered. Discussion Chondromyxoid fibroma is a rare slow-growing bone 8 tumor first described by Jaffe and Lichtenstein in 1943 [1,7,11-13]. Benson and Bass were report first spinal case [14]. Chondromyxoid fibroma is responsible for fewer than 1% of primary bone tumors. More than 500 cases of cmf have ever been described in the world literature. Most of chondromyxoid fibroma (around 75%) occurs metaphysis of long tubular bones. Half of chondromyxoid fibroma is localized to femur and tibia. Chon- dromyxoid fibroma may extend into diaphysis or even rarely epiphysis. It can be purely diaphyseal, but it is 9 never a pure epiphyseal lesion. Mainly second and third decades of life young patients effected [15-17]. Chon- dromyxoid fibroma exceptionally located in the spinal area. In this scarcity mostly found in thoracic spine. Ac- cording to up to date literature lumbar vertebral area is least affected. At the day this paper published there were 11 cases of lumbar chondromyxoid fibroma -re ported (Table 1). Even if some reports have some slight differences there is no gender predominance (Schajo- wicz and H Gallardo). Etiology of the development of Figure 7, Figure 8 and Figure 9: Chondroid areas with chondromyxoid fibroma is unknown [18]. giant cell and fibroblast like cells from our case (H&E X4) (x40, x100, x200 magnifications). Conventional radiography provides the most cost-ef-

KAYA. Neurosurg Cases Rev 2020, 3:043 • Page 3 of 5 • DOI: 10.23937/2643-4474/1710043 ISSN: 2643-4474

Table 1: Literature review of chondromyxoid fibroma of the lumbar spine.

References Age Location Column Symptoms Treatment Follow-up (years)/Sex (Months) Gudscha [2] 23/F L3 Anterior (Body) Back pain; Leg Excision 36, No Weakness Recurrence Ramani, et al. [3] 44/M L1 Pedicle Cauda Equina Excision 2, No Syndrome Recurrence Tsuji, et al. [4] 9/M L4 Posterior (Lamina, Back Pain Excision + Bone 44, No Pedicle) graft Recurrence Mayer [5] 23/M L2 Posterior (Spinous Asymptomatic Biopsy - Processa) Cabral, et al. [6] 19/F L1-L2 Posterior (Pedicle, Leg Pain Extensive Excision 60, Transverse Process) Recurrence Wu, et al. [7] - Three Cases, - - Curettage or - Lumbar Excision Saldua, et al. [8] 8/M L3 Posterior (Pedicle) Back Pain Biopsy; 13, No Excision + Fusion Recurrence Raquel Gutiérrez- 21/M L5 Posterior (Inferior Back Pain Excision 12, No González, et. al. [9] Articular Process) Recurrence Ming-Xiang Zou, et 60/F L1 Anterior and Middle Cauda Equina Excision - al. [10] (Body) Syndrome Present Case 56/F L4 Posteriyor (lamina) Radiculopathy Excision 6, No recurrence fective diagnostic information of any imaging modality. Microscopically, growth is nodular with a high de- However complex structure and adherent neighbors gree of cellularity in the periphery of the nodules. Pro- of spine limits its usefulness. Unless contraindicated duction of either chondroid or fibrocartilaginous matrix Magnetic resonance imaging (MRI) recommended over may be observed. The cellular foci consist of either spin- computed tomography before surgery. Conventional dle-shaped, fibroblast like cells or more myxoid, stel- radiographs show a well-marginated, expansile, lucent, late cells. Mitoses are infrequent. Calcification is found eccentric, medullary lesion [19]. Lesions margins usually microscopically in approximately one fourth of the pa- appear sclerotic and rarely contain visible calcification tients. These patients usually old or lesions are near [15]. Computed tomography scan gives more detailed the bone surface. The extracellular matrix is abundant, information about chondromyxoid fibroma. Primarily myxoid connective tissue and chondroid material. Giant used for MRI contraindicated patients and to identi- cells are frequent. Occasionally, the degree of cellulari- fy cortical integrity. Contrast enhancement is variable ty and nuclear atypia approaches that characteristic of depends on lesion fibroid components and vascularity. malignancy. However, confusion with chondrosarcoma Magnetic resonance imaging the best all-around imag- in usually based on the appearance of the peripheral. ing modality that shows extends of the lesion. Magnet- Microscopically aneurysmal bone cysts, simple bone ic resonance imaging shows broad array of differences cysts, non-ossifying fibroma, giant cell bone tumor and about nature of lesion. In general, low signal intensity chondrosarcoma must consider in mind for differen- on T1-weighted images and heterogenic high signal in- tial diagnosis. Typical view can easily distinguish. Since tensity on T2-weighted images. Also, sclerotic rim and chondrosarkoid transformation at risk, special attention calcification may result hypo intense rim at T2-weighted must be paid for atypia [19]. The stellate cells show fea- images. Small lesions as well as some of larger lesions tures of both chondroblastic and stellate fibroblastic may demonstrate homogenous high signal intensity. differentiation on electron microscopy [18]. Histologic Contrast enhancement images have heterogeneous sig- features don’t predict outcome [13,21,22]. Pericentro- nal intensity [20]. metric inversion; inv (1) (p25q13) is suggested diagnostic marker fort his tumor [15]. According to Sawyer, et Radiologically chondrosarcoma, osteoblastoma, gi- al. 6q13-21 chromosomal abbreviation is found respon- ant cell tumor of bone metastasis, Multiple myeloma, sive local aggressive behavior. hemangioma, and aneurismal bone cysts must be considered in mind [20]. Since, spinal involvement of this rare tumor is even rarer treatment modalities and long term follow up is Macroscopically, chondromyxoid fibroma ranges dictated by surgeon’s experiences. Hence, there is no white to grayish white and the maintains the lobular established protocol. En-block resection is the best growth pattern. The rim may show bone formation. Cys- treatment of choice [11,13,18]. If not possible radical tic and hemorrhagic areas may mimic those seen aneu- curettage and corticocancellous bone grafting must be rysmal bone cyst.

KAYA. Neurosurg Cases Rev 2020, 3:043 • Page 4 of 5 • DOI: 10.23937/2643-4474/1710043 ISSN: 2643-4474 done [13,18,21]. Studies showed en-block resection 4. Tsuji H, Otsuka Y, Tamaki T, Takada N (1975) Chondro- have lowest recurrence rates on the other hand curet- myxoid fibroma of vertebra. Report of a case. J Jpn Orthop Assoc 49: 305-312. tage alone have highest (as much as 80%) recurrence rates [7,12,13,18]. So, if curettage is feasible surgical op- 5. Mayer BS (1978) Chondromyxoid fibroma of lumbar tion bone grafting must be added [13,22]. Also, accord- spine. J Can Assoc Radiol 29: 271-272. ing to Zilmer and Dorfman curettage alone leaves tumor 6. Cabral CEL, Romano S, Guedes P, Nascimento A, Noguei- lobules behind [22]. There are other surgical techniques ra J, et al. (1997) Chondromyxoid fibroma of the lumbar spine. Skeletal Radiology 26: 488-492. such as intralesional excision, or marginal resection [4,21,22]. All other techniques have higher recurrence 7. Wu CT, Inwards C, O’laughlin S, Rock M, Beabout J, et al. (1998) Chondromyxoid fibroma of bone: A clinicopathologic rates. review of 278 cases. Human Pathol 29: 438-446. In case of recurrence radical treatment advocated 8. Saldua NS, Riccio AI, Cassidy JA (2008) Chondromyxoid due to tumor nature and possibility of malignant trans- fibroma of the lumbar spine in a pediatric patient. Orthope- formation. Since, radical excision is treatment principle dics 31: 610. surgical procedure depends on attached area, patient 9. Gutierrez-Gonzalez R, De Reina L, Saab A, Jimenez-Hef- demographic and tumor invasion. fernan J, Garcia-Uria J (2011) Chondromyxoid fibroma of the lumbar spine: Case report and literature review. Eur In our case we made en-block resection and curet- Spine J 21: 458-462. tage. Since surgery 6 month follow up shows no recur- 10. Zou MX, LV GH, Li J, Wang XB (2016) Acute cauda equina rence. syndrome secondary to chondromyxoid fibroma of the lumbar spine. The Spine Journal 16: e587-e588. Conclusion 11. Feldman F, Hecht HL, Johnston AD (1970) Chondromyxoid Chondromyxoid fibroma is rare bone tumor even fibroma of bone. Radiology 94: 249-260. rarer spinal involvement. Because of, malignant trans- 12. Rahimi A, Beabout JW, Ivins JC, Dahlin DC (1972) Chon- formation possibility radical removal is treatment of dromyxoid fibroma: A clinicopathologic study of 76 cases. choice and chondromyxoid fibroma always consider in Cancer 30: 726-736. mind. Surgical treatment depends on attached area, 13. Unni KK (1996) Dahlin’s bone tumors: General Aspects and patient demographic, and tumor invasion. Since, tumor Data on 11,087 cases. American Journal of Clinical Pathol- has a recurrence reports even if complete removal close ogy. follow up must be warranted. More work must be put 14. Benson WR, Bass S (1995) Chondromyxoid fibroma first to understand nature of this rare subject. report of occurrence of this tumor in the vertebral column. Am J Clin Pathol 25: 1290-1292. Acknowledgements 15. Granter SR, Renshaw AA, Kozakewich HP, Fletcher JA Special thanks for my bellowed wife Damla KAYA un- (1998) The pericentromeric inversion, inv(6)(p25q13), is questionable support. Also, I am grateful to Cumhuriyet a novel diagnostic marker in chondromyxoid fibroma. Mod Pathol 11: 1071-1074. University Hospital Department of Pathology for provid- ing slides. 16. Ralph LL (1962) Chondromyxoid fibroma of bone. J Bone and Joint Surg 44B: 7-24. Conflict of Interest 17. Schutt PG, Frost HM (1971) Chondromyxoid fibroma. Clin Orthop Relat Res 78: 323-329. Author doesn’t have any potential conflict of interest. This research did not receive any specific grant from 18. Schajowicz F, Gallardo H (1971) Chondromyxoid fibroma funding agencies in the public, commercial, or not-for- (fibromyxoid chondroma) of bone: A clinico-pathological study of thirty-two cases. J Bone Joint Surg Br 53: 198-216. profit sectors. 19. Vigorita VJ (2016) Orthopedic pathology in cartilage tu- References mors. Wolters Kluver, (3rd edn), Philadelphia, 427-488. 1. Jaffe HL, Lichtenstein L (1948) Chondromyxoid fibroma of 20. White PG, Saunders L, Orr W (1996) Chondromyxoid fibro- bone. A distinctive bone tumor likely to be mistaken espe- ma. Skeletal Radiol 25: 79-81. cially for chondrosarcoma. Arch Pathol 45: 541-551. 21. Gherlinzoni F, Rock R, Picci P (1983) Chondromyxoid fi- 2. Gudscha A (1968) A case of chondromyxoid fibroma of the broma. The experience at the instituto ortopedico Rizzoli. J spine. Ortop Traum Protez 29: 50-52. Bone Joint Surg Am 65: 198-204. 3. Ramani PS (1974) Chondromyxoid fibroma: A rare cause 22. Zilmer DA, Dorfman HD (1989) Chondromyxoid fibroma of of spinal cord compression. J Neurosurg 40: 107-109. bone: Thirtysix cases with clinicopathologic correlation. Hu- man Pathol 20: 952.

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